FETAL DISRUPTIONS - ASSESSMENT OF FREQUENCY, HETEROGENEITY, AND EMBRYOLOGIC MECHANISMS IN A POPULATION REFERRED TO A COMMUNITY-BASED STILLBIRTH ASSESSMENT PROGRAM

被引:43
作者
LUEBKE, HJ
REISER, CA
PAULI, RM
机构
[1] UNIV WISCONSIN, CTR CLIN GENET, MADISON, WI 53705 USA
[2] UNIV WISCONSIN, DEPT PEDIAT, MADISON, WI 53705 USA
[3] UNIV WISCONSIN, DEPT MED GENET, MADISON, WI 53705 USA
来源
AMERICAN JOURNAL OF MEDICAL GENETICS | 1990年 / 36卷 / 01期
关键词
amnion disruption; amniotic bands; fetal death; limb-body wall disruption; stillbirth; twin-twin disruptions;
D O I
10.1002/ajmg.1320360113
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
The Wisconsin Stillbirth Service Project (WiSSP) is a community-based program for the investigation of the cause of fetal death. From its inception in 1983 through July 1988, 629 referrals were made to WiSSP. All referrals were assessed for the presence of disruptional characteristics, and 23 were found to have major or primary disruptive effects. Most of these were either early amnion disruption/limb-body wall disruption (treated as a single group, since analysis suggests a continuum of clinical characteristics) and twin-twin disruptions. Therefore, disruptions accounted for 3.6% of all referrals (including liveborn and miscarriage referrals) to WiSSP. When only stillborn fetuses are considered, approximately 2.4% appear to have died because of disruptions. This makes disruptions one of the most frequent, identifiable causes of late intrauterine death. We estimate that 0.6-1.4% of all stillborn fetuses die because of early amnion disruption/limb-body wall disruption which, when taken with previous estimates of the frequency of such problems in early miscarriages and liveborn infants, suggests that these disruptions result in a 95% prenatal mortality rate. We suggest a unified model of likely pathogenetic mechanisms which may help explain the continuum of multisystem involvement seen in those with early amnion disruption/limb body wall disruption. In addition, 3 patients with atypical disruptions are reviewed who exemplify the difficulty and importance of differentiating disruptional and malformational processes.
引用
收藏
页码:56 / 72
页数:17
相关论文
共 41 条
[1]   CONGENITAL RING CONSTRICTIONS AND INTRAUTERINE AMPUTATIONS [J].
BAKER, CJ ;
RUDOLPH, AJ .
AMERICAN JOURNAL OF DISEASES OF CHILDREN, 1971, 121 (05) :393-&
[2]   ACARDIAC ANOMALY [J].
BENIRSCHKE, K ;
DESROCHESHARPER, V .
TERATOLOGY, 1977, 15 (03) :311-316
[3]  
BENIRSCHKE K, 1967, HDB PATHOL HISTOL, V7, P187
[4]   GENETIC-STUDIES OF AN ACARDIAC MONSTER - EVIDENCE OF POLAR BODY TWINNING IN MAN [J].
BIEBER, FR ;
NANCE, WE ;
MORTON, CC ;
BROWN, JA ;
REDWINE, FO ;
JORDAN, RL ;
MOHANAKUMAR, T .
SCIENCE, 1981, 213 (4509) :775-777
[5]  
BYRNE J, 1982, BIRTH DEFECTS-ORIG, V18, P43
[6]   FAMILIAL CONGENITAL AMPUTATIONS [J].
ETCHES, PC ;
STEWART, AR ;
IVES, EJ .
JOURNAL OF PEDIATRICS, 1982, 101 (03) :448-449
[7]   EPIDEMIOLOGY OF THE EARLY AMNION RUPTURE SPECTRUM OF DEFECTS [J].
GARZA, A ;
CORDERO, JF ;
MULINARE, J .
AMERICAN JOURNAL OF DISEASES OF CHILDREN, 1988, 142 (05) :541-544
[8]   LIMB REDUCTION ANOMALIES AND EARLY INUTERO LIMB COMPRESSION [J].
GRAHAM, JM ;
MILLER, ME ;
STEPHAN, MJ ;
SMITH, DW .
JOURNAL OF PEDIATRICS, 1980, 96 (06) :1052-1056
[9]   ACCURACY OF FETAL DEATH REPORTS - COMPARISON WITH DATA FROM AN INDEPENDENT STILLBIRTH ASSESSMENT PROGRAM [J].
GREB, AE ;
PAULI, RM ;
KIRBY, RS .
AMERICAN JOURNAL OF PUBLIC HEALTH, 1987, 77 (09) :1202-1206
[10]   OCULO-AURICULAR CRANIAL DYSPLASIA [J].
GUPTA, JS ;
GUPTA, SD ;
PRASHAR, SK .
BRITISH JOURNAL OF OPHTHALMOLOGY, 1968, 52 (04) :346-&